Int. J. Oral Surg.

1984: 13: 85-91

(Key words: cyst, aneurysmal bone; cyst, bone; cyst; jaw cyst, surgery, oraf)

Aneurysmal bone cyst of the jaws

(I). Clinicopathological features

P. J. STRUTHERS AND M. SHEAR

Department of Oral Pathology, School of Pathology, University of the Witwatersrand and South
African Institute for Medical Research, Johannesburg, South Africa

ABSTRACT - A clinicopathological study has been performed on a series of

42 well-documented examples of aneurysmal bone cyst of the jaws
recorded in the literature, and 4 additional personally observed cases. Most
occurred in the first three decades (93%), 2/3 of patients being younger
than 20 years. There was a preponderance of females (62%). Both
mandible and maxilla were involved, particularly the molar regions. A
number of mandibular cases extended to the angle and ascending ramus.
Swelling was usually present and there was frequently a history of rapid
growth. Radiologically, they often appeared as multilocular radiolucencies
with expansion and thinning of the cortical plates. Histologically, the
lesions consisted of multiple cystic spaces of varying size, usually filled with
blood. The intervening solid tissue frequently showed features of other
pathological lesions, particularly the central giant cell granuloma, but
occasionally as fibrous dysplasia, ossifying fibroma and cementifying
fibroma. Recurrences have been common probably because of technical
difficulties in entirely removing very large lesions. Thorough curettage, if
necessary by an extraoral approach, is the most favoured method of
treatment.

(Receioed for publication 15 November 1982, accepted 28 March 1983)

The term 'aneurysmal bone cyst' was first
used by JAFFE & LICHTENSTEIN in 194223 .
JAFFE 2 4 pointed out that the adjective
'aneurysmal' used in this context relates to
the 'blowout' distension of part of the con-
tour of the affected bone area, creating the
striking radiographic appearance which is
so often a feature of the lesion. The bony
expansion usually causes visible swelling of
the overlying soft tissues (Figs. I and 2). At
operation, an intact periosteum and very
thin shell of bone usually covers the cyst.
When this is removed, dark venous blood
wells up and bleeding may be difficult to
control. Histologically, the cyst contains
variable amounts of soft tissue, consisting of
friable vascular tissue, which subdivides the
cavity into a number of blood-filled locu-
1es 4 9 • Part of the lesion may contain areas of
more solid tissue4 4 ,4 9 .
Approximately 650 cases involving the
entire skeleton have been reported but the
lesion is regarded as rare in the jaws. We are
aware of only about 42 cases having been
86 STRUTHERS AND SHEAR

reported in the literature. In our depart-

ment, only 5 cases have been accessioned
over the past 21 years, one of which has
been reported previously?".
This study was therefore undertaken in
order to accumulate as much clinicopatho-
logical data as possible about the aneurys-
mal bone cyst of the jaws.

Material and methods

An extensive review of the literature on the
aneurysmal bone cyst of the jaws was undertaken.
42 well-documented cases with sufficient informa-
tion to confirm the diagnosis of aneurysmal bone
cyst were included in the study. The 4 personally
observed cases, not previously reported, were
added to this material. Age, sex, site, clinical
presentation, treatment and histological features
were recorded for each case.
Fig. 2. Radiograph of the lesion shown in Fig. 1,
demonstrating expansion of the cortex and the
multilocularity of the lesion (courtesy of Dr. S.
Butz).

Results
Age and sex
The age and sex distribution of 45 cases are
shown in Fig. 3. Most cases occurred in the
first 3 decades of life (93%), with a peak in
the second decade. 64% of cases were pa-
tients below the age of 20 years. There was a
preponderance of females (28 cases: 62%).

Fig. 1. Clinical photograph of a 5-year-old girl
with an aneurysmal bone cyst occurring in the
right body and angle of the mandible (courtesy of
Dr. S. Butz).
Site
Of the 46 cases, 28 occurred in the mandible
and 18 in the maxilla. One cyst was found
close to the orbital floor and another in the
zygomatic arch. The anterior region of the
mandible was rarely involved. Most cases
involved the molar regions of the mandible
and maxilla, a number of mandibular cases
extending to involve the angle and ascend-
ing ramus.
 ANEURYSMAL BONE CYST
Clinical presentation
In the great majority of cases, obvious
swelling was present. Where the rate of
growth of the swelling was recorded, this
had been relatively rapid in 16 cases and
gradual in 4.
Trauma did not appear to playa signifi-
cant aetiological role. In 13 instances, a
history of some pain was reported, whereas
in another 16 the lesion was painless. In the
remaining cases the presence or absence of
pain was not recorded. Pain, however, does
not appear to be a significant feature of
these lesions and some authors ascribed it to
stretching of the overlying tissues, or to
secondary infection or interference with
temporomandibular joint function.
Radiological features
When radiological features were recorded,
37 lesions were reported as radiolucent,
frequently showing a "soap bubble" or
apparently multilocular cystic appear-
ance21.34.46 with expansion and thinning of
the cortical plates (Fig. 2). Only one lesion
was reported as predominantly radiopaque.
Treatment
Lesions were usually treated by curettage or
resection and the results are included in
30
25
DMAlES
(/J
21
~FEMALES
UJ
~ 20
o
u, 15 10
0 13
~ 10
5
2 were below the age of 30 years. RUITER et
0 --'''2-' 0 0 0
AGE
Fig. 3. Age and sex distribution of 45 aneurysmal
bone cysts of the jaws.
87
Table 1 if a follow-up period of longer than
6 months was recorded.
For jaw cases, some operators preferred
to pack the bone cavity with material such
as bone chips, iodoform or gypsum follow-
ing curettagel,12.28.33,37.
Pathological features
The blood-filled spaces which characterize
the aneurysmal bone cyst were present in all
lesions and serve as a major diagnostic
criterion. Most cases showed the presence of
giant cells of the type seen in the central
giant cell granuloma of the jaws (Table 2).
Other cases demonstrated histological or
clinical features suggesting association of
the aneurysmal bone cyst with fibrous dys-
plasia of bone. Some lesions included areas
which resembled ossifying or cementifying
fibromas histologically. One case had
originally been diagnosed histologically as
an osteosarcoma, although this may have
been a misdiagnosis'", Of the 4 personally
observed cases, which are indicated with
asterisks in Table 2, two showed areas re-
sembling the giant cell granuloma and two
the ossifying fibroma. Photomicrographs of
these lesions are shown in the second paper
of this series'i'',
Discussion
The age and sex distributions are similar to
those reported in series of aneurysmal bone
cysts occurring outside the jaws except that
a smaller proportion of the jaw cases occur
in the under 20 age group. Nevertheless,
there is little doubt that jaw cases occur in
young patients as 93% in the present study
al. 4 1 published a series of 105 cases (only
one of which was located in the jaws) in
which almost 85% of the patients were 20
years or younger. 59 (56%) occurred in
females. BIESECKER et al" studied 66 non-
jaw cases in which 70% of the patients were
88 STRUTHERS AND SHEAR

Table 1. Results of treatment of 19 aneurysmal bone cysts of the jaws, where a follow-up oflonger than
6 months was reported

Method of No. of No. of %

treatment cases recurrences recurrences

curettage 15 8 53
partial resection 4 1 25

younger than 20 years. 39 (59%) were
female. TILLMAN et ai.5 0 described a series
of 95 cases (in which there were only 2 jaw
cases) of which almost 80% were below the
age of 20. 55 (59%) were female.
The frequent occurrence of rapid growth
is of interest in view of the variable histolog-
ical features. The most commonly as-
sociated histological pattern is that of the
central giant cell granuloma and this is not
usually a rapidly enlarging lesion. It is there-
fore possible that the aneurysmal bone cyst
enlarges slowly at first, until erosion of the
cortical plate of bone has occurred, after
which a rapid "blowout" draws attention to
what appears to be a rapidly growing
swelling.
The aneurysmal bone cyst has a higher
recurrence rate 8 •2 0 than the central giant cell
granuloma. The high recurrence rate of
53% for jaw cases following treatment by
curettage is similar to that reported for
larger series of cases occurring in other
bones. BmSECKER et al. 4 reported that 26
of 44 cases (59%) treated by curettage re-
curred. There were 4 recurrences in the 6
cases which CLOUGH & PRICE 1 0 treated by
curettage. TILLMAN et al. 50 had similar expe-
riences with 20 cases, 7 recurring (35%).
The main difficulty in deciding how to
treat the aneurysmal bone cyst would ap-
pear to be that, although the clinicopatho-
logic picture of the aneurysmal bone cyst of
the jaws is well documented, and is very
similar to that of the lesion found outside
the jaws, the pathogenesis and pathological
processes involved in the development of the
lesion are not fully understood. When as-
sessing a case of aneurysmal bone cyst,
careful note of the histopathological fea-
tures of the solid areas within the lesion
should be taken. A considerable variability
of histopathological pattern has been re-
ported as occurring in association with
aneurysmal bone cysts, both in the jaw
(Table 2) and in other bones 4 , 7,9 , 1 0 . 14 ,2 5,
30,48,49, and this may influence the recur-
rence rate of the lesion. Indeed, even mal-

Table 2. Histological features in 43 of the series of 46 aneurysmal bone cysts of the jaws which might
indicate the presence of an associated pathological lesion; the 4 personal cases are indicated by asterisks

Feature No. of cases

1. Giant cells of the type seen in the 321-3.11-13.17-19.26-28.31.33.35-39,43,47,51-53,*.*

central giant cell granuloma

2. Fibrous dysplasia 57 •1 6,2 8 ,2 9 , 3 2
3. Ossifying fibroma 36 .* ,*
4. Cementifying fibroma 2 1 5 •22
5. Osteosarcoma (7) 11 9
 ANEURYSMAL BONE CYST
ignant tissue has occasionally been reported
concurrent with aneurysmal bone oysts".
These latter cases may, however, represent
examples of the telangiectatic osteosar-
coma, which, in a detailed histopathological
study, RUITER et al.4 2 showed to have a very
similar presentation' to that of the aneury-
smal bone cyst. Overall, the aneurysmal
bone cyst is regarded as a benign lesion and
malignant behaviour has very seldom been
reported'': 19,30,45.
The variable pathological features ob-
served in association with aneurysmal bone
cysts have led to speculation that the lesion
develops as a result of secondary change in
another primary lesion 4 , 5. 1 0.4 4 ,4 9 . Many of
the cases studied in the present review
showed areas with striking resemblance to
the central giant cell granuloma, yet the
aneurysmal bone cyst appears to be con-
siderably more difficult to eradicate than
this lesion. The high recurrence rate of the
aneurysmal bone cyst of the jaws following
curettage may possibly be explained by the
technical difficulties which may be experien-
ced in completely eradicating the extensive
cystic lesion, rather than by the biological
behaviour of the tissue itself. Several
authors mention that the surgery was com-
plicated by massive haemorrhage, in many
cases necessitating ligation of the external
carotid art ery 1 3 , 1 6. 1 9 . 3 1 ,3 2 . 3 3 ,3 9 . Adequate
curettage of lesions involving the mandi-
bular angle-ramus area necessitates expo-
sure by an extraoral approach. BIESECKER et
al," reduced the recurrence rate by supple-
menting curettage with cryotherapy. Sup-
plementation of curettage by radiotherapy
has also been used for ajaw case'": TILLMAN
et at. 50 have, however, reported sarcoma-
tous change following radiotherapy, and
this type of treatment is definitely not re-
commended except in rare cases where the
aneurysmal bone cyst may be secondary to a
malignant neoplasm''". Supplementation of
curettage by cautery has also been attem-
89
pted, but without success>". In conclusion,
we believe that thorough curettage, with
careful follow-up is the treatment of choice
for the aneurysmal bone cyst of the jaws.
The pathological characteristics of the
specimen should be carefully studied, as the
presence of an associated aggressive lesion
may necessitate modification of this ap-
proach.
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Address:
M. Shear
Department of Oral Pathology
University of Witwatersrand
1, Jan Smuth Avenue
Johannesburg
South Africa